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Home , Ankylosing spondylitis, Arthritis mutilans, Sacroiliitis

Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Ann. rheum. Dis. (1979), 38, Supplement p. 12

Radiological manifestations of Reiter's syndrome

WILLIAM MARTEL From the University of Michigan Hospital, Michigan, USA

Reiter's syndrome (RS) presents with a broad Table 1 Distribution of and radiological spectrum of radiological manifestations. Although features in 36 cases of Reiter's syndrome there is an overlap with other connective tissue Distribution No. ofcases diseases134, 214, 221, 248, 286 certain features appear to of upper extremities 11 be characteristic, particularly when present in Joints of lower extremities 30 combination. This report concerns the radiological Forefoot 18 Heel 18 findings in 36 patients with RS. They are compared Interphalangeal great toe 8 with the radiological manifestations of rheumatoid Feet more than hands 16 arthritis (RA), ankylosing (AS), and Sacroiliac arthritis 26 Symmetric 17 (PSA). Asymmetric 6 Unilateral 3 Spondylitis 16 Patients and methods Atypical for 14 Typical for ankylosing spondylitis 2 Focal 4 by copyright. Records of 36 patients were selected from the files of Periosteal apposition the department of , University of Michigan (appendicular joints) 26 Medical Center, on the basis of previously docu- mented positive radiological findings. The records were then reviewed to confirm the clinical diagnosis of RS. For the purpose of this study the diagnostic affected in only 11. The most common site of criteria were (1) objective evidence of arthritis of the involvement was the foot, particularly the metatarso- appendicular joints, and (2) two of the following: phalangeal joints and heels. The posterosuperior and mucocutaneous lesions, conjunctivitis, and urethritis. posteroinferior aspects of the calcaneus were These features had to have occurred within six affected (Figs 1, 2), particularly the latter, at the weeks of one another. It was recognised that the attachment of the plantar aponeurosis. The inter- http://ard.bmj.com/ arthritis in patients with RS may be limited to the phalangeal of the great toe was affected in eight spine, but because of the difficulty in evaluating such instances and this appeared to be relatively selective arthritis, particularly in the early stages, such cases in three (Fig. 3). Occasionally there was severe were not included in the study. destruction of some metatarsophalangeal joints but Radiological joint surveys of the spine and adjacent ones seemed completely spared. The hips appendicular joints were available in 20 cases. Six were affected in one case bilaterally. Arthritis was others had had fairly complete radiological evalua- more extensive in the feet than the hands in 16 cases. on September 29, 2021 by guest. Protected tions, although not all joints were included. Films of The terminal interphalangeal joints of the hands were the lumbosacral spine were available in all cases. affected in two patients and only one such joint was Many patients had been followed-up clinically for affected in both. The sacroiliac joints were affected in several years, affording opportunities for extended 26 patients and the spinal column in 16. Focal periods ofradiological observation. sacroiliitis near a sarcoiliac joint was noted in four cases. Radiological features REGIONAL Significant radiological features are summarised in This was a striking finding in the early stages of the Table 1. disease in three patients. It was limited to the foot in two cases (Fig. 4) and to the hand in one. A frequent DISTRIBUTION feature was relative absence of osteoporosis despite Joints of the lower extremities were affected in 30 extensive joint destruction. This was particularly cases whereas joints of the upper extremities were evident in the foot. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

L Features andprognosis Suppl. p. 13

Soft-tissue swelling, presumably non-specific due narrowing of the interosseous space. Bone destruc- to joint effusion and synovitis, was common in the tion occasionally appeared as discrete marginal , knee, and joints of the hand, wrist, and foot. erosions within the joint at the chondro-osseous junctions ('bare areas') (Fig. 5) or as loss of cortical DESTRUCTION OF ARTICULAR CARTILAGE definition within and adjacent to the joints. Sites of This was common and manifested by uniform were often blurred by coexistent by copyright.

Fig. 1 Progressive clacaneal erosion. Early erosion in May 1971 (arrow) was more extensive in September 1971 wit,A blurred margins due to associated minimal reactive bone presumably formation. http://ard.bmj.com/ on September 29, 2021 by guest. Protected

Fig. 2 Note erosion at posterosuperior aspect of Fig. 3 Relatively selective arthritis ofinterphalangeal calcaneus (arrow) and extensive periosteal bone joint ofgreat toe with severe joint destruction and apposition at inferior aspects ofcalcaneus, especially reactive bone formation. Note soft tissue swelling of posteriorly, and cuboid. Calcaneocuboidjoint 4th and 5th toes with erosion ofdistal interphalangeal relatively intact but cuboidmetatarsaljoint affected. joint ofthe 4th. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Suppl. p. 14 Annals of the Rheumatic Diseases

Fig. 4 Osteoporosis, right foot, associated with minimal generalised soft-tissue swelling. by copyright. http://ard.bmj.com/ on September 29, 2021 by guest. Protected

Fig. 5 Arthritis limited to proximal interphalangeal K...: ::::.: ::: :- .-- joint. Note fusiforn soft-tissue swelling, uniform loss of cartilage, and marginal erosions. Erosions partially Fig. 6 Severe destructive arthritis and subluxation of obscured by periosteal bone apposition. multiple joints with sparing of4th metatarsophalangeal joint. Note involvement of interphalangeal joint of great toe and relative lack ofosteoporosis. periosteal bone apposition. Large subchondral PERIOSTEAL BONE APPOSITION cyst-like lesions were not seen. Severe destructive This was seen in the appendicular skeleton in 24 arthritis with extensive and sub- cases, was often exuberant and fluffy during the luxation ('' or Launois's deformity) active stage of , and was typically was observed in three cases. All involved the contiguous to affected joints (Fig. 7). Later in the metatarsophalangeal joints. One or more joints may disease such bone characteristically appeared linear be 'skipped'-that is, completely spared (Fig. 6). and compact, giving the cortex a 'thickened' Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

L Features andprognosis Suppl. p. 15

appearance (Fig. 8). Focal bone apposition, adjacent to a previously affected joint, was at times the only indicator of prior inflammation. This feature was often subtle (Fig 9).

SACROILIAC ARTHRITIS This occurred in 26 cases and was symmetrical in 17. In the latter it was indistinguishable from the radiological changes of ankylosing spondylitis (Fig. 10). The appearance depended on the stage of the disease, with the early lesion often appearing as an indistinctness ofthe subchondral cortices (Fig. 11). Reactive sclerosis in the adjacent bone varied in degree. Bc!nv was common. In six patients sacroiliac arthritis was clearly asymmetric and in three it was unilateral. In three cases the asymmetry or unilaterality persisted for several years (Fig. 12). In four patients there was a focal sclerosis and hyperostosis, involving both and contiguous

Fig. 7 Arthritis offirst metatarsophalangealjoint associated with bone erosion andperiosteal bone by copyright. apposition ofmetacarpal and, to lesser extent, phalanx (arrows). Note minimal uniform narrowing of interosseous space ofthis joint. http://ard.bmj.com/ on September 29, 2021 by guest. Protected

~~~~~~~~~~~~~ Fig. 8 Destructive arthritis, right fourth metatarsophalangealjoint, with deformity and widening ofbones due to periosteal bone apposition in a child. Note involvement offirst tarsometartasaljoint with marginal bone erosions andlack ofosteoporosis in right foot. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Suppl. p. 16 Annals of the Rheumatic Diseases

ilium, adjacent to the cranial margin of the joint (Fig. 13). This appeared to be analagous to the hyperostoses in the appendicular skeleton and vertebral bodies.

SPONDYLITIS Two of the 16 cases with spinal involvement showed changes indistinguishable from those of ankylosing spondylitis. However, the others had a distinctive appearance characterised by asymmetric bony bridges between contiguous vertebral bodies, usually involving the lateral aspects (Fig. 14). These often resembled massive syndesmophytes, appearing to arise from the vertebral body proper, and often there were several such bone formations which were non-continuous. The vertebral body margins where these arose occasionally showed deformity and sclerosis, suggesting that the bone formation reflected vertebral rather than inflamma- tion of the discovertebral joints. Furthermore, the intervertebral discs were often ofnormal height at the affected levels. Although there was squaring of the vertebral by copyright. bodies in some cases the anterior surfaces of the vertebrae tended to be spared, even at those levels where lateral bony bridging was present. In some Fig. 9 Subtle periosteal bone apposition, right medial patients segments of the spine appeared to be malleolus-a residuum of earlier arthritis of the ankle 'skipped'-for example, in one case the spondylitis in which at this time appeared normal. Compare with the cervical segment was fairly advanced whereas the opposite normal side. dorsal spine:andfmuch of the lumbar segment http://ard.bmj.com/ on September 29, 2021 by guest. Protected

IOA Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

I. Features andprognosis Suppl. p. 17 by copyright. lOB lCC

Fig. 10 A. Bilateral sacroiliac arthritis with reactive sclerosis. B. Asymmetric lateral bony bridge between Li and L2, having appearance oflarge syndesmophyte. C. Anterior vertebral surface relatively normal and height ofdisc not reduced at this level. No other lumbar spinal lesions present. Although the sacroiliac arthritis in this case was indistinguishable from that seen in ankylosing spondylitis the lumbar lesion is characteristic ofRS and PSA.

appeared to be spared (Fig. 15). Two cases showed http://ard.bmj.com/ unilateral, vertically-oriented ossification adjacent to a discovertebraljoint (Fig. 16). The adjacent vertebral body was unaffected and the appearance suggested that this lesion was related to the lateral spinal ligament. In no cases of spondylitis was there sparing of the sacroiliac joints, but 10 patients with

sacroiliac arthritis did not exhibit spondylitis. on September 29, 2021 by guest. Protected Differential diagn9sis

RHEUMATOID ARTHRITIS (RA) Although erosions occur at the chondro-osseous junctions ('bare areas') in RS this distribution is more conspicuous in RA and the erosions appear more discrete. The feet may occasionally be affected before the hands in the early stages of RA, but it is unusual to see significantly greater involvement in Fig. 11 Early right sacroiliac arthritis in RS. The the feet than the hands as the disease progresses; lesion consists ofan indistinctness of the cortices, predilection for the joints of the lower extremities is .---particularly------1 on--- the-- iliac-- - side.---- unusual in RA. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Suppl. p. 18 Annals of the Rheumatic Diseases

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12 At on September 29, 2021 by guest. Protected E;ig. 12 A. 1958. B. 1964. Persistent asymmetric sacroiliac arthritis: left side more severely involved than right.

Periosteal bone apposition occurs in juvenile RA phalangeal joint of the great toe is not a feature of adjacent to affected joints, but it is almost always RA. linear rather than fluffy and exuberant. Significant The sacroiliac joints are rarely affected in RA, but periosteal bone apposition of any type is unusual in the cartilage space has been reported on occasion to adult RA. Subchondral cyst-like lucencies (pseudo- have become completely obliterated. Sclerosing cysts) are not uncommon in and osteiitis in the sacroiliac joints and bone proliferation tend to be multiple, but they are apparently rare in at the vertebral body margins, however, are decidedly RS. Asymmetric arthritis does occur in adult RA but unusual. The spondylitis in RA is most evident it is uncommon and not associated with other radiologically in the cervical region and is features of RS. Selective involvement of the inter- characterised by bone destruction and subluxation. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

I. Features andprognosis Suppl. p. 19 by copyright. Fig. 13 Focal sclerosing osteitis above right in 40-year-old man with RS. Lesion appears to involve contiguous sacroiliac margins. Left sacroiliacjoint normat, right equivocal. There were no lesions in the lumbar spin3. _

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14A 14B Fig. 14 A. 1963. Large, asymmetric bony bridge L3-L4 and bilateral sacroiliac arthritis in man with RS. B. 196g. Sacroiliac joints now fused andprevious bony bridge altered owing to remodelling. Other syndesmophytes now evident at L2-3 and L1-2, but generalised syndesmophyte formation as generally seen in ankylosing spondylitis has not developed. Note fairly well preserved disc heights. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Suppl. p. 20 Annals of the Rheumatic Diseases by copyright. 15A 15B Fig. 15 A. 1963. B. 1968. Same case as in Fig. 14, showing advanced involvement of cervical spine. Dorsel spine was not affected. This case illustrates the tendency towards skipped segments and non-uniform spondylitis. A similar distribution may be seen in PSA. http://ard.bmj.com/ on September 29, 2021 by guest. Protected

---...... -. - _ Fig. 16 Paravertebral ossification which is lateral, adjacent to disc, but separate from vertebrae. Such ossification has also been described in PSA. Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

I. Features andprognosis Suppl. p. 21

The latter is particularly common in the atlantoaxial be affected in PSA, and often many such joints are joints. Subluxation ofthe atlantoaxial joints has been involved. Although patients with RS occasionally described in RS197 but it is rare. It is doubftul have arthritis of a terminal interphalangeal joint whether true syndesmophyte formation occurs in RA. multiple involvement of such joints is rare. Resorp- tion of the terminal tufts of the fingers occasionally ANKYLOSING SPONDYLITIS (AS) occurs in PSA but this is not a feature of RS. Although the spondylitis in RS is occasionally Periosteal bone apposition adjacent to affected identical to that of AS most RS patients show joints is common in PSA and is often fluffy and distinctive features. These include focal syndes- exuberant, as in RS. Similarly, the calcaneal changes mophyte formation, often massive and associated and the tendency to involve the interphalangeal joint with little change in the adjacent discovertebral of the great toe are common in both diseases. The joints; relative sparing of the anterior surfaces of the feet may be more severely affected than the hands in vertebral bodies; and a tendancy towards non- PSA, but severe involvement of the feet with uniform involvement of the spinal column (often relatively little change in the hands seems to be more with'skipped' segments). common in RS. Panarthritis of the hands and In most cases of RS the sacroiliac arthritis is in wrists is not uncommon in PSA but is distinctly rare itself indistinguishable from that seen in AS. in RS. Nevertheless, asymmetric or unilateral involvement of the sacroiliac joints should prompt suspicion that Discussion one is not dealing with ankylosing spondylitis. Focal sclerosing osteitis adjacent to the sacroiliac joint Patients were included in this study because they had proper may be a significant differential point which had abnormal radiological joint findings earlier. has not been emphasised. There is a predilection for Therefore the features described here probably by copyright. the rhizomelic joints in AS, whereas in RS the hips reflect a more severe form of the disease. The and shoulders are not commonly affected. observations are significant, however, in that thay Irreversible structural changes, including articular indicate the spectrum of radiological findings that bone erosion, may develop in the hands and feet in may be encountered in RS. Since the patient sample AS, but usually this is a late manifestation. As in RS, was small and the radiological joint survey incom- there may be periosteal bone apposition near plete in many conclusions about the precise incidence affected joints and marginal erosions at the chondro- of specific features are probably not warranted. osseous junctions, but mutilating destruction of the Although none of the described features are in is unusual. Heel lesions may be identical in themselves pathognomonic ofRS certain ones should both conditions but extensive bone destruction and strongly suggest this diagnosis, particularly when http://ard.bmj.com/ widespread periosteal bone apposition of the present in combination. These are listed in Table 2. calcaneus is unusual in ankylosing spondylitis. The significance of focal paravertebral ossification, Selective involvement of the interphalangeal joint of seen in two cases, is uncertain, This appears to lie in the great toe is not a feature ofAS. relation to the lateral ligament and differs from syndesmophytes in that it does not seem to involve PSORIATIC ARTHRITIS (PSA) the vertebrae. Bywaters and Dixon49 described

There is a significant overlap between the radiological similar lesions in four patients with PsA but in three on September 29, 2021 by guest. Protected features of PA and RS. This is particularly true of the of their cases the possibility of RS was raised. spondylitis. As in RS, some patients with PA show a Asymmetric spondylitis with a tendency to 'skipped' pattern which is identical to that seen in AS. But in segments is characteristic though not diagnostic. A most cases it is more like that of RS, including similar pattern has been observed in PSA.208 221 In a asymmetrical involvement of the sacroiliac joints, large non-continuous syndesmophytes primarily involving the lateral aspects of the vertebral bodies Table 2 Radiologicalfeatures characteristic of with relative sparing of the anterior surfaces, and Reiter's syndrome 'skipped' segments. (1) Severe involvement of the feet with relative sparing of the hands (2) Predilection for the calcaneus, interphalangeal joint of the I have never seen focal sacroiliitis adjacent to the great toe, and metatarsophalangeal joints sacroiliac joint proper in PSA, but it is not clear (3) Periosteal bone apposition near affected joints whether this will prove to be a differential (4) Sacroiliac arthritis, especially when asymmetric significant (5) Asymmetric, often large, bridging syndesmophytes involving point. An important differential feature is the tendency mainly the lateral aspects of the vertebral bodies with relative for the terminal interphalangealjoints ofthe fingers to sparing of their anterior surfaces Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

Suppl. p. 22 Annals of the Rheumatic Diseases recent survey of clinical and radiographic one with Felty's syndrome. This incidence is to be abnormalities in 87 patients with AS it was concluded expected in view of the 1-2 % population prevalence that the pattern of spinal involvement differed in of RA. men and women.255 DR. E. ALBERT: We have observed a family in which, Focal sclerosing sacroiliitis, seen in four cases within one generation, four siblings were homo- adjacent to the sacroiliac joint, is of interest. zygous for B27 with apparently classical RA. In the Increased density due to may be noted in second generation all the children were heterozygous this location in of the sacroiliac joints. for B27, and four or five of them had developed AS. However, our patients were relatively young and Has anyone information about the full genotyping or showed no other features of osteoarthritis in these also the phenotyping of such patients that seem to joints. To my knowledge this has not been noted in have both AS (or RS) and RA? PSA, RA, or AS but additional studies are needed DR. D. A. BREWERTON: In looking at families of before diagnostic significance is attached to this patients with AS it is all too common to find previous finding. generations that have been wrongly diagnosed as RA. When they are reinvestigated clearly they have a Conclusion form of related to B27 and spondylitis. PROF. T. BITTER: I know that Professors Vischer and The radiological manifestations of RS overlap those Fallet from Geneva have systematically tissue-typed of other rheumatic diseases, particularly AS and a large series of patients with RA for B27 and got a PSA. Nevertheless, certain features are characteristic slightly higher percentage of B27 than expected and, depending on the combination of findings in a in the general population. Would they want to given case, the diagnosis may be suspected on comment on the clinical features of these B27 radiographical grounds. rheumatoids? by copyright. PROF. G. H. FALLET: The problem has presented itself General Discussion slightly differently. Beyond three of Dr. Mason's patients in London and one in Oxford, we have DR. J. T. SCOTT: What do you consider the best observed in Geneva six patients who presented with radiological projection for sacroiliac joint evalu- both apparently seropositive RA and AS. Three of ation ? them had rheumatoid nodules identified by biopsy. PROF. MARTEL: I don't advocate oblique views because Two patients had children, one child of each had AS. they are often confusing and hide early lesions. I All but one of them had HLA-B27 and x-ray findings with the with prefer to have an angled beam patient compatible both diseases. Michael Mason's and http://ard.bmj.com/ supine (some radiologists prefer the patient prone). our feeling is that unless these patients had a The beam is angled 150-200 towards the head. This is coincidental predisposition for both diseases we effect distorts the sacroiliac joints but shows the should revise our concept ofRA and AS. inferior aspect of the sacroiliac area, which is the PROF. A. E. GOOD: From our small arthritis clinic we true joint. reported three patients in 1977 with classical findings DR. G. R. V. HUGHES: We and others have seen typical for both diseases. In summing up the reports about patients with both seropositive erosive RA and patients with RA, nodules, and AS we found that B27-positive sacroiliitis. Have you looked at the neither disease seemed to interfere with the expression on September 29, 2021 by guest. Protected small but definite percentage ofrheumatoids who are of the other. It seems to us that the association is B27-positive to determine whether B27 confers a purely accidential and not so rare after all. different pattern on coincidental RA ? PROF. R. F. WILLKENS: Dr. Martel would you comment PROF. C. M. PEARSON: We have seen cases ofcoinciding on a non-marginal syndesmophyte and the distinction spondylitis and seropositive rheumatoid arthritis ofAS from RS on the basis ofthese? with nodules which were B27-positive. Cases reported DR. MARTEL: I haven't used the terms 'marginal' and so far have been tabulated in the April 1978 issue of 'non-marginal' syndesmophyte. The syndesmophytes Arthritis and . However, the pattern of in RS and in PSA may be identical to those in AS- RA seems to have been quite classical as well as that the so-called 'marginal' ones. However, the large ofAS ,either beginning first. asymmetric syndesmophytes characteristically seen DR. F. c. ARNETT: Within nearly 200 patients with in RS are indeed so large that they appear to be either RS or AS we have three patients with non-marginal. They seem to develop as a conse- coinciding seropositive nodular rheumatoid arthritis, quence of vertebral osteitis and bone formation Ann Rheum Dis: first published as 10.1136/ard.38.Suppl_1.12 on 1 January 1979. Downloaded from

I. Features and prognosis Suppl. p. 23

thereafter, not necessarily related to the disco- rapidly over a period of six to twelve weeks. Have vertebral junctions. Whenever they are present they you any studies on sequential change? are useful in differentiating RS from AS. I have not DR. MARTEL: No, I have not. seen such bone formation in classical AS. The DR. J. C. GERSTER: Achilles tendonitis is found very sacroiliac joints are the least help unless the sacroiliac commonly in RS. In a study with Professor Fallet arthritis is asymmetrical. Such asymmetry can be we found it in AS, PSA, and especially in RS. We did seen early in the course of AS. It is unusual to find not find it in RA. severe involvement on one side as the disease DR. MARTEL: We have seen achilles tendonitis in progresses with little or no change on the other. rheumatoid arthritis. Radiologically it is often Most of the time patients with RS present with different from the three conditions you mentioned. sacroiliac arthritis which is indistinguishable from DR. T. L. VISCHER: I was struck that you have very that of AS. little joint narrowing in your RS patients. PROF. B. AMOR: With the former we found more DR. MARTEL: I agree that the cartilage often seems to destructive lesions. Likewise bone proliferation on be less affected than the bone. There is often bone the great toes seems to be more common in psoriatic erosion and bone apposition with relatively little than in RS. change in the interosseous space. The cartilage may DR. MARTEL: The appearance I have shown in the feet be altered and yet the interosseous space is not is identical to what we may see in PSA, including the narrowed. The space does not reflect the degree of heels and terminal interphalangeal joints of the great cartilage destruction because the fluid and soft tissue toes. In AS I am not sure. We have seen patients with proliferation may take its place within the joint. AS with severe arthritis of the hands and feet as a DR. CALIN: We have not emphasised the entheso- late manifestation-a picture indistinguishable from pathic nature of the disease. Clinically we recognise that seen in RS. In the latter, however, structural sausage digits, insertional tendinitis, and other by copyright. changes in the joints of the feet often occur relatively extrasynovial processes. These correlate with early. radiological evidence of periostitis, peri-insertional DR. A. CALIN: What is the rate of progression of these osteoporosis, and other changes that differentiate lesions? We have had the impression that some this enthesopathic disorder from the primary patients with RS have developed plantar spurs synovitides, typified by RA. http://ard.bmj.com/ on September 29, 2021 by guest. Protected